Psarras et al. Journal of Medical Case Reports 2011, 5:463 JOURNAL OF MEDICAL http://www.jmedicalcasereports.com/content/5/1/463 CASE REPORTS

CASEREPORT Open Access Amyand’s -a vermiform appendix presenting in an : a case series Kyriakos Psarras, Miltiadis Lalountas*, Minas Baltatzis, Efstathios Pavlidis, Anastasios Tsitlakidis, Nikolaos Symeonidis, Konstantinos Ballas, Theodoros Pavlidis and Athanassios Sakantamis

Abstract

Introduction: A vermiform appendix in an inguinal hernia, inflamed or not, is known as Amyand’s hernia. Here we present a case series of four men with Amyand’s hernia. Case presentations: We retrospectively studied 963 Caucasian patients with inguinal hernia who were admitted to our surgical department over a 12-year period. Four patients presented with Amyand’s hernia (0.4%). A 32-year-old Caucasian man had an inflamed vermiform appendix in his hernial sac (acute appendicitis), presenting as an incarcerated right groin hernia, and underwent simultaneous and Bassini suture . Two patients, Caucasian men aged 36 and 43 years old, had normal appendices in their sacs, which clinically appeared as non-incarcerated right groin . Both underwent a plug-mesh hernia repair without appendectomy. The fourth patient, a 25-year-old Caucasian man with a large but not inflamed appendix in his sac, had a plug-mesh hernia repair with appendectomy. Conclusion: A hernia surgeon may encounter unexpected intraoperative findings, such as Amyand’s hernia. It is important to be prepared and apply the appropriate treatment.

Introduction often pose technical dilemmas, even for the experienced A vermiform appendix in an inguinal hernia sac, with or surgeon [2]. The surgeon may encounter unusual find- without appendicitis, is called Amyand’s hernia. Clau- ings, such as a vermiform appendix partly or fully con- dius Amyand (1660-1740), a French surgeon working at tained in the hernia sac, inflamed or non-inflamed, St George’s and Westminster hospitals in London, per- stretched or curved, and adhered or not adhered to the formed the first successful appendectomy in 1735, on an sac walls. Whether or not an appendectomy should be 11-year-old boy who presented with an inflamed, perfo- performed at the same times as the hernia repair is rated appendix in his inguinal hernia sac. According to debatable. The aim of this study is to present the experi- the surgeon’s descriptions, the patient also had “a fistula ence of our university surgical department with between the scrotum and thigh” and the operation Amyand’s hernias along with a review of the literature proved to be “very complicated and perplexing,” as the on this subject. pathology consisted of a chronically inflamed appendix contained within the inguinal hernia sac, perforated by a Case presentations previously swallowed pin. At surgery the appendix was We undertook a retrospective review of the case his- removed. The patient eventually recovered and was “dis- tories of 963 Caucasian patients with inguinal hernia, charged with a truss, which he was ordered to wear for admitted and treated in our surgical department over a some time.” The case was published in the Philosophical 12-year period (between 1998 and 2009). Both elective Transactions of the Royal Society of London [1]. and emergency cases were included in the study. Infor- Inguinal hernia repair is one of the most common mation was obtained from their medical records and operations in surgical practice. Despite that, hernias their detailed operative protocols. Four Caucasian patients presented with Amyand’s hernia (0.4%). All * Correspondence: [email protected] patients had an uneventful postoperative course, without 2nd Propedeutical Department of Surgery, Hippokration Hospital, Medical School of Aristotle, University of Thessaloniki, Greece

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any recorded postoperative wound infection or hernia favored by many authors [9,10]. Appendectomy adds the recurrence. risk of infection to an otherwise clean procedure. Super- ficial wound infection increases morbidity; and deep Case 1 infection may contribute to hernia recurrence. In addi- A 32-year-old man, with the clinical appearance of an tion, surgical manipulation to achieve visualization of incarcerated right groin hernia, had acute appendicitis the entire appendix and its base, by enlarging the her- and underwent simultaneous appendectomy and con- nial defect or distending the neck of the hernial sac, ventional modified Bassini hernia repair. increases the possibility of recurrence by weakening the anatomic structures around the defect [2,5,7,10]. There Case 2 are authors who recommend reduction of the appendix A 36-year-old man, with the clinical appearance of a and mesh hernioplasty if there is no acute appendicitis, non-incarcerated right groin hernia, with a normal and appendectomy followed by endogenous hernia appendix within his hernia sac, had a mesh-plug hernia repair if an inflamed appendix is found [7,10,11]. repair without appendectomy. Although these general rules are certainly acceptable, there are more clinical scenarios to keep in mind. Case 3 Losanoff and Basson have distinguished four basic types A 43-year-old man, with the clinical appearance of a of Amyand’s hernias, which should be treated differently non-incarcerated right groin hernia, with a normal (see Table 1 for classification) [4,5]. appendix within his hernia sac, had a mesh-plug hernia The absence of inflammation in Type 1 advocates elec- repair without appendectomy. tive hernioplasty. Using a prosthetic material in such cases carries the expectation of improved longevity of the Case 4 repair. It avoids tension on the suture lines and circum- The fourth patient, a 25-year-old man, presented with a vents the metabolic problems related to collagen defi- 20 cm long but non-inflamed appendix. This was fully ciency, which is known to exist in hernia patients. contained and adhering to his inguinal sac wall, pulling Whether to remove or leave behind a normal appendix the cecum up to the internal inguinal orifice. The in this clinical scenario cannot be determined because no patient underwent a mesh-plug hernia repair along with evidence-based information exists. The decision is rather appendectomy. based on common sense, relating to the patient’sage,life expectancy, life-long risk of developing acute appendicitis Discussion and the size and overall anatomy of the appendix. Pedia- Acute appendicitis within an inguinal hernia accounts tric or adolescent patients have a significantly higher risk for 0.1% of all cases [2-7]. Inflammation of the appendix of developing acute appendicitis and should therefore is attributed to external compression of the appendix at have their appendices removed, compared to middle- the neck of the hernia. The inflammatory status of the aged or elderly individuals in whom the appendix should vermiform appendix determines the surgical approach probably be left intact [4,5]. Long, curved appendices and the type of hernia repair. All surgeons agree that if have a higher risk of inflammation. Additionally a long appendicitis exists, the repair of the hernia should be appendix which stretches the cecum may cause chronic performed with Bassini or Shouldice techniques, without pain if left behind. Manipulations to detach and reduce making use of synthetic meshes or plugs within the the appendix in the abdomen may stimulate the inflam- defect [2,5,8] due to the high risk of suppuration of matory process. Furthermore, consideration of appen- such materials. dectomy in young patients must take into account the In the case of a normal appendix, incidentally found size of the hernia, since prosthetic material is contraindi- within the hernia sac, the performance of a prophylactic cated but large hernias are more likely to recur if repaired appendectomy along with the hernia repair is not by making use of endogenous tissue only.

Table 1 Classification of Amyand’s hernias after Losanoff and Basson [4,5] Classification Description Surgical Management Type 1 Normal appendix within an inguinal hernia Hernia reduction, mesh repair, appendectomy in young patients Type 2 Acute appendicitis within an inguinal hernia, no abdominal sepsis Appendectomy through hernia, primary endogenous repair of hernia, no mesh Type 3 Acute appendicitis within an inguinal hernia, abdominal wall or , appendectomy, primary repair of hernia, no mesh peritoneal sepsis Type 4 Acute appendicitis within an inguinal hernia, related or unrelated Manage as types 1 to 3 hernia, investigate or treat second abdominal pathology pathology as appropriate Psarras et al. Journal of Medical Case Reports 2011, 5:463 Page 3 of 3 http://www.jmedicalcasereports.com/content/5/1/463

The decision is easier in Type 2 hernias, where appen- References 1. Amyand C: Of an inguinal rupture, with a pin in the appendix caeci, dicitis is found, as they should be treated with appen- incrusted with stone; and some observations on wounds in the guts. dectomy; however the hernia repair should be Philos Trans R Soc London 1736, 39:329-336. performed without making use of prosthetic materials. 2. Ballas K, Kontoulis T, Skouras C, Triantafyllou A, Symeonidis N, Pavlidis T, ’ Marakis G, Sakadamis A: Unusual findings in inguinal hernia surgery: On the other hand, in septic patients with Amyand s Report of 6 rare cases. Hippokratia 2009, 13(3):169-171. hernia Type 3 (acute appendicitis with peritonitis), or 3. De Garengeot RJC: Traite des operations de chirugie. 2 edition. Paris: Huart; Type 4 (acute appendicitis with other pathology), even 1731, 369-371. – the hernioplasty may be contraindicated if the patient’s 4. Losanoff JE, Basson MD: Amyand hernia: what lies beneath a proposed classification scheme to determine management. Am Surg 2007, condition is poor or life expectancy is limited. 73(12):1288-1290. Looking at our case series, in case 1 we decided not to 5. Losanoff JE, Basson MD: Amyand hernia: a classification to improve place a mesh, due to the presence of acute inflamma- management. Hernia 2008, 12(3):325-326. 6. Llullaku SS, Hyseni HS, Kelmendi BZ, Jashari HJ, Hasani AS: A pin in tion–appendicitis. This guarded the hernia repair from appendix within Amyand’s hernia in a six-years old boy: case report and possible future extension of inflammation in the mesh. review of literature. World J Emerg Surg 2010, 5:14. ’ In contrast, a mesh was placed in cases 2 and 3 with a 7. Milanchi S, Allins AD: Amyand s hernia: history, imaging, and management. Hernia 2008, 12(3):321-322. normal appendix in their sac. However, in these cases, 8. Livaditi E, Mavridis G, Christopoulos-Geroulanos G: Amyand’s hernia in we decided not to proceed with appendectomy, because premature neonates: report of two cases. Hernia 2007, 11(6):547-549. ’ this additional procedure could lead to potential damage 9. Sharma H, Gupta A, Shekhawat NS, Memon B, Memon MA: Amyand s hernia: a report of 18 consecutive patients over a 15-year period. Hernia of the plastic hernia repair. In case 4, given the young 2007, 11(1):31-35. age of our patient and the long appendix in his sac, we 10. D’Alia C, Lo Schiavo MG, Tonante A, Taranto F, Gagliano E, Bonanno L, Di ’ decided that the increased likelihood for appendicitis in Giuseppe G, Pagano D, Sturniolo G: Amyand s hernia: case report and review of the literature. Hernia 2003, 7(2):89-91. the future necessitated an individual appendectomy. 11. Salemis NS, Nisotakis K, Nazos K, Savrinou P, Tsohataridis E: Perforated Consequently, our recommendation is that the decision appendix and periappendicular abscess within an inguinal hernia. Hernia to perform an appendectomy or/and use the mesh-plug 2006, 10(6):528-530. technique should always be individualized to the patient. doi:10.1186/1752-1947-5-463 Cite this article as: Psarras et al.: Amyand’s hernia-a vermiform appendix presenting in an inguinal hernia: a case series. Journal of Medical Case Conclusion Reports 2011 5:463. In conclusion, a hernia surgeon may encounter unex- pected intraoperative findings, such as an Amyand’s her- nia. The decision as to whether one should perform a simultaneous appendectomy and hernia repair is multi- factorial. It is important to be aware of all clinical set- tings and an appropriate and individualized approach should be applied.

Consent Written informed consent was obtained from all patients for publication of this case series and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Authors’ contributions KP, KB, TP performed the procedures. ML obtained the patients’ written informed consent to publish the report, conducted the follow-up examinations, analyzed and interpreted the patient data, and wrote part of Submit your next manuscript to BioMed Central the manuscript. KP, NS, MB, EP and AT edited and wrote part of the and take full advantage of: manuscript. KB and TP were major contributors to reviewing and editing the manuscript. AS made the strategic plan and gave the final approval. All • Convenient online submission authors read and approved the final manuscript. • Thorough peer review Competing interests • No space constraints or color figure charges The authors declare that they have no competing interests. • Immediate publication on acceptance

Received: 29 April 2011 Accepted: 19 September 2011 • Inclusion in PubMed, CAS, Scopus and Google Scholar Published: 19 September 2011 • Research which is freely available for redistribution

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